An 81 year old man with a history of hypertension received intravenous recombinant tissue plasminogen activator (tPA) for right middle cerebral artery (MCA) infarction. He had not had stroke or arrhythmia previously. The initial National Institute of Health Stroke Scale (NIHSS) score was 8. However, a left MCA territory infarction developed 2 minutes after the full course of tPA therapy was completed, and 24 hours after tPA infusion, NIHSS score was 17. The subsequent magnetic resonance imaging scan confirmed an extensive left MCA territory infarction and a small right MCA territory infarction. Although the intracerebral haemorrhage after tPA therapy is relatively more common, tPA infusion may result in an ischaemic cerebral stroke in rare cases.